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Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis

To assess the efficacy and safety of human leukocyte antigen–matched allogeneic cultivated limbal epithelial stem cell grafts in the treatment of aniridia-associated keratopathy (AAK). METHODS: Six eyes of 6 patients with severe AAK received an allogeneic stem cell graft between January 2010 and Mar...

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Autores principales: Behaegel, Joséphine, Tassignon, Marie-José, Lagali, Neil, Consejo, Alejandra, Koppen, Carina, Ní Dhubhghaill, Sorcha
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cornea 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8647694/
https://www.ncbi.nlm.nih.gov/pubmed/33928920
http://dx.doi.org/10.1097/ICO.0000000000002729
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author Behaegel, Joséphine
Tassignon, Marie-José
Lagali, Neil
Consejo, Alejandra
Koppen, Carina
Ní Dhubhghaill, Sorcha
author_facet Behaegel, Joséphine
Tassignon, Marie-José
Lagali, Neil
Consejo, Alejandra
Koppen, Carina
Ní Dhubhghaill, Sorcha
author_sort Behaegel, Joséphine
collection PubMed
description To assess the efficacy and safety of human leukocyte antigen–matched allogeneic cultivated limbal epithelial stem cell grafts in the treatment of aniridia-associated keratopathy (AAK). METHODS: Six eyes of 6 patients with severe AAK received an allogeneic stem cell graft between January 2010 and March 2017. Anatomical and functional results were assessed at 6 months, 1 year, 2 years, and the final follow-up visit available. Safety analysis was performed by considering all perioperative and postoperative adverse events and additional surgeries required during the follow-up period. RESULTS: The mean follow-up was 53.6 months (range 24–104 months). In most patients (80%), there was an early improvement of the keratopathy postoperatively, which slowly regressed during longer follow-up. At the final follow-up, 4 of the eyes were graded as failure and 1 eye was graded as partial success. Grading the sixth eye was not possible because of an adverse event. None of the patients maintained a total anatomical success in the long-term. Only 1 patient maintained a modest improvement in best-corrected visual acuity from hand motion to counting fingers. Four serious adverse events were recorded in 2 patients. CONCLUSIONS: Severe AAK remains a challenging condition to manage. Transplantation of allogenic ex vivo cultivated limbal stem cells may provide a temporary improvement in ocular surface stability, but anatomical and functional results are poor in the long-term. The eyes are prone to adverse events, and any surgical treatment should take this into consideration.
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spelling pubmed-86476942021-12-07 Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis Behaegel, Joséphine Tassignon, Marie-José Lagali, Neil Consejo, Alejandra Koppen, Carina Ní Dhubhghaill, Sorcha Cornea Clinical Science To assess the efficacy and safety of human leukocyte antigen–matched allogeneic cultivated limbal epithelial stem cell grafts in the treatment of aniridia-associated keratopathy (AAK). METHODS: Six eyes of 6 patients with severe AAK received an allogeneic stem cell graft between January 2010 and March 2017. Anatomical and functional results were assessed at 6 months, 1 year, 2 years, and the final follow-up visit available. Safety analysis was performed by considering all perioperative and postoperative adverse events and additional surgeries required during the follow-up period. RESULTS: The mean follow-up was 53.6 months (range 24–104 months). In most patients (80%), there was an early improvement of the keratopathy postoperatively, which slowly regressed during longer follow-up. At the final follow-up, 4 of the eyes were graded as failure and 1 eye was graded as partial success. Grading the sixth eye was not possible because of an adverse event. None of the patients maintained a total anatomical success in the long-term. Only 1 patient maintained a modest improvement in best-corrected visual acuity from hand motion to counting fingers. Four serious adverse events were recorded in 2 patients. CONCLUSIONS: Severe AAK remains a challenging condition to manage. Transplantation of allogenic ex vivo cultivated limbal stem cells may provide a temporary improvement in ocular surface stability, but anatomical and functional results are poor in the long-term. The eyes are prone to adverse events, and any surgical treatment should take this into consideration. Cornea 2022-01 2021-04-22 /pmc/articles/PMC8647694/ /pubmed/33928920 http://dx.doi.org/10.1097/ICO.0000000000002729 Text en Copyright © 2021 The Author(s). Published by Wolters Kluwer Health, Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND) (https://creativecommons.org/licenses/by-nc-nd/4.0/) , where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.
spellingShingle Clinical Science
Behaegel, Joséphine
Tassignon, Marie-José
Lagali, Neil
Consejo, Alejandra
Koppen, Carina
Ní Dhubhghaill, Sorcha
Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis
title Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis
title_full Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis
title_fullStr Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis
title_full_unstemmed Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis
title_short Outcomes of Human Leukocyte Antigen–Matched Allogeneic Cultivated Limbal Epithelial Transplantation in Aniridia-Associated Keratopathy—A Single-Center Retrospective Analysis
title_sort outcomes of human leukocyte antigen–matched allogeneic cultivated limbal epithelial transplantation in aniridia-associated keratopathy—a single-center retrospective analysis
topic Clinical Science
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8647694/
https://www.ncbi.nlm.nih.gov/pubmed/33928920
http://dx.doi.org/10.1097/ICO.0000000000002729
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